Eur J Cardiothorac Surg 2005;28:495-496
© 2005 Elsevier Science NL
Off pump coronary artery bypass grafting in a patient with tracheopathia osteoplastica
Mohan P. Devbhandari
a
,
*
,
Tahir Mirza
a
,
Michael Hartley
b
,
Andrew J. Duncan
a
a Department of Cardiothoracic Surgery, Blackpool Victoria Hospital, Blackpool FY3 8NR, UK
b Department of Anaesthesia, Blackpool Victoria Hospital, Blackpool, UK
Received 12 November 2004;
received in revised form 21 March 2005;
accepted 23 March 2005.
* Corresponding author. Tel.: +44 1253 655833; fax: +44 1254 303669. (Email: sdevbhandari{at}aol.com).
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Abstract
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Tracheopathia osteoplastica is a rare disease of the tracheobronchial tree, which is often misdiagnosed and recognised only at post-mortem. We report the case of a 66-year-old man with this condition, mistakenly thought to be suffering from chronic bronchiectasis. He underwent successful coronary artery bypass grafting for unstable angina using an off-pump technique. A case report and brief literature review is presented.
Key Words: Tracheopathia osteoplastica • Benign • Bronchial tree
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1. Introduction
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Tracheopathia osteoplastica (TO) is a benign disease of the tracheobronchial tree characterised by numerous nodules of bone and cartilage protruding into the lumen from the wall of the respiratory tree. We report a case where we unexpectedly encountered this condition whilst attempting endotracheal intubation in preparation for urgent coronary artery bypass grafting (CABG) for unstable angina. The patient had mistakenly been thought to be suffering from bronchiectasis for a number of years. A summary of the case is presented along with a literature review.
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2. Case report
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A 66-year-old man with chronic stable angina following a myocardial infarction 10 years previously was referred with crescendo angina. Past history revealed repeated episodes of deep vein thrombosis and long standing bronchiectasis. Coronary risk factors were hyper-cholesterolemia and family history of ischaemic heart disease. The coronary angiogram showed significant stenosis of the left anterior descending artery and the circumflex artery. At preoperative assessment he was found to be symptomatic with frequent episodes of productive cough, which resulted in postponement of his operation by few weeks. A computerised tomographic (CT) scan of the chest at this time was reported to show changes of chronic obstructive airway disease with bronchiectasis.
However, while waiting for surgery he developed unstable angina and he was referred for urgent in-patient surgical revascularisation. In theatre, general anaesthesia including muscle relaxation was induced uneventfully following which controlled ventilation was established via an oropharyngeal airway. Direct laryngoscopy revealed normal laryngeal anatomy and tracheal intubation was attempted. The tracheal tube passed easily through the vocal cords but could not be advanced into the trachea due to the presence of an obstructive lesion. A laryngeal mask airway was inserted and fiberoptic bronchoscopy performed. This revealed diffuse brown hard nodules covering the entire length of trachea, carina, and main-stem bronchi up to lobar bronchi, sparing the posterior membranous portion (Fig. 1
).
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Fig. 1. Bronchoscopic view of airway showing ‘rock garden’ appearance due to multiple submucosal nodules with sparing of membranous portion posteriorly.
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To our unaware mind, this finding raised a strong suspicion of malignancy. Multiple biopsies were taken and the operation was postponed. Contrary to our expectations the biopsy result did not show malignancy but merely chronic inflammatory changes. A literature search enabled us to identify this lesion to be tracheopathia osteoplastica. The treating respiratory physicians had mistaken this lesion for bronchiectasis. Retrospective review of the CT scan showed the prominent abnormality in the trachea. The trachea showed a double ring, with an outer complete ring corresponding to the anatomical tracheal wall, and the inner incomplete ring (due to sparing of membranous portion) created by the tracheopathia osteoplastica.
A further joint discussion with cardiology favoured CABG to a percutaneous intervention. It was felt that the use of an OPCAB technique would give him the best chance of a long-term result with acceptable short-term risks. At operation intra-thecal morphine was administered prior to anaesthesia to provide postoperative pain relief, followed by induction of general anaesthesia and intermittent positive pressure ventilation via a laryngeal mask airway. The operation was uneventful. He was awake and the LMA was removed immediately on arrival in the intensive care unit. He was well enough to be transferred to the ward on the first postoperative day and was discharged home on the sixth postoperative day.
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3. Discussion
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The first macroscopic [1] and microscopic [2] descriptions of TO were published in 1855 and 1857 respectively. The exact etiology of this condition is not known. This was thought to be due to ecchondroses and exostoses by Virchow [3], whereas Aschoff [4] proposed that it was initiated by metaplasia of elastic tissue in internal elastic fibrous membrane. Others have suggested it to be due to chronic inflammation, leading to accumulation of calcium salts within the tracheal mucosa around which cartilage and bone gradually develop [5].
In a series of 30 cases over 12 years, average age at presentation was 42 and 51 years in males and females, respectively [5]. Although most of the patients have some symptoms like cough, wheezing, hoarseness or haemoptysis, these are usually non-specific and are often mistaken for other conditions. Most cases remain undiagnosed, with only 5% of cases being correctly diagnosed before death [6]. The bronchoscopic appearance of ‘cobblestone’ or ‘rock garden,’ usually in the lower third of trachea is diagnostic of this condition. Our patient, unusually, had extensive involvement starting from the larynx and going into the lobar bronchi as far as the fiberoptic bronchoscope could visualise. Multiple submucosal nodules are well-recognised diagnostic radiological features of TO [7]. In keeping with Young and colleagues findings in their postmortem pathological study [8], the CT scan of our patient showed presence of ‘double concentric rings’, an inner nearly complete ring of calcified nodules and outer ring corresponding to anatomical airway.
There are only two reported cases of TO undergoing CABG in literature. In the first case, TO was encountered as an incidental finding at induction [9]. The patient was intubated with an 8-mm endotracheal tube, and surgery was performed uneventfully using cardiopulmonary bypass (CPB). The postoperative period was complicated by an episode of chest infection, which resolved with treatment. In the second case, the disease was much more localised [10]. A rigid bronchoscope was passed allowing insertion of a bougie, followed by a microlaryngoscopy tube for ventilation. Surgery was performed with CPB. He was electively returned to theatre the next day for planned extubation. Obstructing nodules were removed piecemeal with a CO2 laser first, followed by an uneventful extubation.
This is the first case in the literature of a patient with this rare abnormality undergoing OPCAB. In spite of extensive disease with bronchoscopic evidence of involvement of the whole extent of visible tracheobronchial tree, the use of intrathecal opioid analgesia and laryngeal mask anaesthesia allowed us to extubate the patient at the end of the procedure while maintaining excellent pain relief and the ability to cough effectively to clear secretions.
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4. Conclusion
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Using OPCAB with laryngeal mask airway, surgery can be safely conducted, for this rare condition.
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References
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- Rokitansky K cited by Dalgaard JB. Tracheopathia osteoplastica. Acta Pathol Microbial Scand 1947;24:118-134.
- Wilks S. Ossific deposits on the larynx, trachea and bronchi. Trans Pathol Soc 1857;8:88.
- Virchow R (1863) cited by Sakula A. Tracheobronchopathia osteoplastica. Thorax 1968;23:105–10..
- Aschoff L. Uber tracheopathia osteoplastica. Verh Dtsch Pathol Ges 1910;14:125-127.
- Harma RA, Suurkari S. Tracheopathia chondro-osteoplastica. A clinical review of thirty cases. Acta Otolaryngol 1977;84:118-123.[Medline]
- Secrest PG, Kendig TA, Beland AJ. Tracheobronchopathia osteochondroplastica. Am J Med 1964;36:815-818.[Medline]
- Bottles K, Nyberg DA, Clark M, Hinchcliffe WA. CT diagnosis of tracheobronchopathia osteochondroplastica. J Comput Assist Tomogr 1983;7:324-327.[Medline]
- Young RH, Sandstrom RE, Mark GJ. Tracheopathia osteoplastica clinical, radiologic, and pathological correlations. J Thorac Cardiovasc Surg 1980;79:537-541.[Abstract]
- Smith DC, Pillae R, Gillbe CE. Tracheopathia osteochondroplastica a cause of unexpected difficulty in tracheal intubation. Anaesthesia 1987;42:536-538.[Medline]
- Birzgalis AR, Farrington WT, O'Keefe L, Shaw J. Localized tracheopathia osteoplastica of the subglottis. J Laryngol Otol 1993;107:352-353.[Medline]
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Abstract
1. Introduction
